Download Hiccup: An Extremely Rare Presentation of Thyrotoxicosis of Graves

Oman Medical Journal (2011) Vol. 26, No. 2: 129-130
DOI 10. 5001/omj.2011.32
Hiccup: An Extremely Rare Presentation of Thyrotoxicosis of Graves’ Disease
Feroze Ahmed, Mohammed Ashraf Ganie, Nasir Shamas, Mohammad Wani, Irshad Parray
Received: 09 Jan 2011 / Accepted: 03 Feb 2011
© OMSB, 2011
Abstract
Case Report
Persistent hiccup is a rare but potentially severe condition that can
be symptomatic of a variety of diseases or idiopathic. Most episodes
last only a few minutes and are self-limited, but hiccup can get
persistent and become a real problem for physician and patient alike.
The center of hiccup may be activated by a great variety of stimuli
travelling along different nerve pathways and bring different effecter
responses. We report a case of persistent hiccup as a presentation of
impending thyroid storm of Graves’ disease. Though the condition
is rare, clinicians should remain alert to the possibility of this
diagnosis.
A 55 years-old male, chronic smoker, shopkeeper by occupation
diagnosed case of Graves’ disease with moderate ophthalmopathy
was on our follow-up for the last 12 months. He was stabilized
on carbimazole 20 mg a day for two months before the current
admission, when he defaulted with the medication. He started with
persistent and non responding hiccup for 10 days (i.e. two months
after drug withdrawal).
The frequency and intensity of hiccups worsened with time and
were almost present throughout the day and night at the time of
admission to the emergency room of the hospital. There was no
history of drug intake known to cause hiccups, abdominal pain, and
chest pain, as well as symptoms pointing to a mass or sepsis in the
chest or abdomen.
On examination, the patient was ill looking, confused,
disoriented, diaphoretic and moderately emaciated with a BMI
of 19.4 Kg/m2. Vitals in ER revealed moderate fever (103.5°F),
severe tachycardia (pulse of 122 bpm), and a wide pulse pressure
(BP 130/60 mm Hg). He had diffuse, soft to firm thyromegaly
with bruit over it. Eye examination revealed that both were
hyperadrenergic and mechanical (bilateral proptosis) components
of ophthalmopathy were indicative of activity. The rest of the
systemic examination was unremarkable.
Investigations revealed normal blood counts, urinalysis,
electrocardiography, liver and kidney functions. Electrogastrodudenoscopy, chest roentgenography, Ultrasonography (USG) of
the abdomen, Contrast Enhanced Computed Tomography (CECT)
of the brain were normal. USG of the thyroid showed diffuse
enlargement with no nodules. Thyroid functions tests revealed very
high tri-iodothyronine and L thyroxine levels with undetectable
TSH. (Table 1)
Keywords: Persistent hiccup; Graves’ disease; Impending thyroid
storm; Thyrotoxicosis.
Introduction
C
hronic hiccup is defined as an attack lasting longer than 48
hours and, sometimes it can be serious, intractable and exhausting.
Persistent Hiccup (PH) is rare and it can be idiopathic or a symptom
of a variety of diseases. The hiccup center may be activated by a
great variety of stimuli travelling along different nerve pathways and
a reflex arc has been proposed, with the phrenic nerves, vagi, and
T6-12 sympathetic fibers as the afferent limb.1 The "hiccup centre"
is thought to be located in either the brainstem respiratory centre or
the cervical cord between segments C3 and C5.2 The efferent limb
is the phrenic nerve.3 It has been suggested that hiccups could be a
form of myoclonus, caused by repeated and abnormal activity of the
solitary inspiratory nucleus.4 We present a case of Graves’ disease
which had defaulted with drug treatment and presented with PH.
The patient had impending thyroid storm and its treatment resulted
in remission of hiccup. We hypothesize that hyperthyroxinemia
caused the irritation of the hiccup centre that resolved after taking
care of hyperthyroxinemia. This case highlights a rare presentation
of severe thyrotoxicosis.
Feroze Ahmed, Mohammed Ashraf Ganie
, Nasir Shamas,
Irshad Parray
Department of Endocrinology
Sheri Kashmir Institute of Medical Sciences, Srinagar J&K, India.
Email: [email protected] / [email protected]
Mohammad Wani
Department of Neurology. Sheri Kashmir Institute of Medical Sciences, Srinagar
J&K , India.
Table 1: Serial thyroid function tests
Investi- Zero 1st
gation week week
3rd
week
10th
week
Normal range
T3
4.04
1.41
1.03
00.70 - 02.50ng/ml
T4
12.71 12.13
11.25 9.28
05.50 - 13.50 ug/dl
TSH
<0.001 <0.001 <0.001 <0.001
1.82
00.50 - 06.50UIu/ml
The patient was treated for impending thyroid storm with
external cooling, intravenous fluids, and carbimazole 60 mg/day in
Oman Medical Specialty Board
Oman Medical Journal (2011) Vol. 26, No. 2: 129-130
divided doses, 100 mg of IV hydrocortisone eight hourly and beta
blockers. The clinical condition improved and the hiccups decreased
in frequency and intensity, until they stopped on the third day of
admission. On enquiry, the patient gave similar history of mild
hiccup at the first diagnosis of Graves’ disease which had resolved
after anti-thyroid treatment. He was discharged on carbimazole
after two weeks of inpatient care and is currently euthyroid and
under consideration for radioiodine ablation as definitive therapy.
(Fig. 1)
monitoring and manometry should be included in the diagnostic
evaluation systematically.
Most episodes of hiccup are associated with acute gastric
distention and alcohol ingestion, but these are short lived and resolve
spontaneously.6 Etiological treatment is not always available and
chronic hiccup treatment has classically relied on metoclopramide
and chlorpromazine. Recently, baclofen has emerged as a safe and
often effective treatment.5 To the best of our knowledge; this is the
first case which highlights hiccup as one of the rare presentations of
thyrotoxicosis.
In this report, we attempted to rule out most of the usual causes
of hiccup and most of the evaluation was negative. We assume that
irritation of the hiccup centre by high levels of thyroxine resulted in
persistent hiccup since it was the treatment of thyrotoxicosis, which
resulted in its resolution. It was not the steroid effect as he continued
with hiccups even on steroids, and by day three, the hiccups stopped
completely, which also corresponds to the normalization of serum
T3 levels. Also, the fact that the patient had no recurrence of the
symptoms in the euthyroid state and no other cause was evident after
a long follow up of 18 months further confirmed our hypothesis.
Conclusion
Figure 1: USD Thyroid showing diffuse enlargement of the thyroid.
Discussion
Hiccups are an involuntary spasmodic and coordinated contractions
of the inspiratory muscles associated with a delayed and sudden
closure of the glottis which is responsible for the characteristic
noise.1
Hiccups are a common benign disorder, which are usually
transient. Occasionally, hiccups fail to resolve spontaneously,
resulting in patient fatigue, incapacitation and the need for the
affected individual to seek medical care in order to resolve the
problem.
The approach to the management of these patients consists of
the identification and treatment of serious underlying causes of
the episode as well as therapeutic interventions to achieve hiccup
resolution. Chronic Persistent hiccup is defined as one lasting more
than 48 hours, which is uncommon, and Graves’ disease presenting
with hiccup is rare.5 It can be serious, intractable, and exhausting,
and its cause often obscure. It may indicate multiple neurologic and
extra neurologic disorders such as brain tumor, strokes, myocardial
infarction, renal failure, prostate cancer, or abdominal surgery.
Psychogenic causes (excitement, stress) may also elicit hiccups.
Detailed medical history and physical examinations will often guide
diagnostic investigations (abdominal ultrasound, chest or brain
CT scan). Gastric and duodenal ulcers, gastritis, esophageal reflux
and esophagitis are commonly observed in chronic hiccup patients
and upper gastrointestinal investigations such as; endoscopy, pH
Oman Medical Specialty Board
We report this case to highlight this rare presentation of thyroid
storm/severe thyrotoxicosis. It emphasizes to practitioners that it
is important to thoroughly investigate a case of hiccup to identify
any underlying pathological cause with due consideration to
hyperthyroxinemia/thyroid storm as in this case.
Acknowledgements
The authors reported no conflict of interest and no funding was received
on this work.
References
1. Noble EC. Hiccup. Can Med Assoc J 1934 Jul;31(1):38-41.
2. Salem MR, Baraka A, Rattenborg CC, Holaday DA. Treatment of hiccups
by pharyngeal stimulation in anesthetized and conscious subjects. JAMA
1967 Oct;202(1):126-130.
3. Baraka A. Inhibition of hiccup by pulmonary inflation. Anesthesiology 1970
Mar;32(3):271-273.
4. Moretti R, Torre P, Antonello RM, Ukmar M, Cazzato G, Bava A.
Gabapentin as a drug therapy of intractable hiccup because of vascular lesion:
a three-year follow up. Neurologist 2004 Mar;10(2):102-106.
5. Launois S, Bizec JL, Whitelaw WA, Cabane J, Derenne JP. Hiccup in adults:
an overview. Eur Respir J 1993 Apr;6(4):563-575.
6. Moretti R, Torre P, Antonello RM, Nasuelli D, Cazzato G. Treatment of
chronic: new perspectives. Eur J Neurol 1999 Sep;6(5):617.